Osteopoikilosis is an autosomal dominant benign sclerosing bony dysplasia, typically found incidentally on imaging studies done for other orthopedic reasons ^1-4. The radiographic and CT features are multiple benign enostoses ^1-4.  Its importance is predominantly to recognize this condition so that it is not mistaken for pathology and prevent unnecessary emotional distress and invasive testing ^2,4.

 This case demonstrates an atypical presentation of osteopoikilosis diagnosed incidentally, with involvement of the skull and spine.